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Proper limb development requires concerted communication between cells within the developing limb bud. Several molecules have been identified which contribute to the formation of a circuitry loop consisting in large part of secreted proteins. The intracellular actin nucleator, Formin 1 (Fmn1), has previously been implicated in limb development, but questions remain after the identification of a Gremlin transcriptional enhancer within the 3′ end of the Fmn 1 locus. To resolve this issue, a knockout mouse devoid of Fmn1 protein was created and characterized. The mice exhibit a reduction of digit number to four, a deformed posterior metatarsal, phalangeal soft tissue fusion as well as the absence of a fibula to 100% penetrance in the FVB genetic background. Importantly, this mutant allele does not genetically disrupt the characterized Gremlin enhancer, and indeed  Gremlin  RNA expression is upregulated at the 35 somite stage of development. Our data reveal increased Bone Morphogenetic Protein (Bmp) activity in mice which carry a disruption in  Fmn1 , as evidenced by upregulation of  Msx1  and a decrease in  Fgf4  within the apical ectodermal ridge. Additionally, these studies show enhanced activity downstream of the Bmp receptor in cells where  Fmn1  is perturbed, suggesting a role for Fmn1 in repression of Bmp signaling.

Formin1 disruption confers oligodactylism and alters Bmp signaling